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Communicating the diagnosis of psychogenic nonepileptic seizures: The patient perspective
Journal of Clinical Neuroscience xxx (2016) xxx–xxx
Contents lists available at ScienceDirect
Journal of Clinical Neuroscience journal homepage: www.elsevier.com/locate/jocn
Clinical Study
Communicating the diagnosis of psychogenic nonepileptic seizures: The patient perspective Amir Arain a,⇑, Maamoon Tammaa a, Faria Chaudhary a, Shazil Gill a, Syed Yousuf a, Nandakumar Bangalore-Vittal a, Pradumna Singh a, Shagufta Jabeen b, Shahid Ali b, Yanna Song a, Nabil J. Azar a a b
Department of Neurology, Vanderbilt University Medical Center, A-0118 Medical Center North, Nashville, TN 37232, USA Department of Psychiatry, Meharry Medical College, Nashville, TN, USA
a r t i c l e
i n f o
Article history: Received 27 February 2015 Accepted 4 October 2015 Available online xxxx Keywords: Epilepsy Nonepileptic seizures Pseudoseizures Psychogenic seizures Spells
a b s t r a c t Psychogenic nonepileptic seizures (PNES) are a common cause of refractory seizures. Videoelectroencephalographic (EEG) monitoring has allowed PNES to be effectively distinguished from epileptic seizures. Once the diagnosis of PNES is established, neurologists face the challenge of explaining it to patients. Patients may not always receive the diagnosis well. The aim of this study is to evaluate how effectively patients receive and perceive the diagnosis of PNES. This prospective study was conducted in an eight-bed epilepsy monitoring unit (EMU). Adult patients with newly confirmed PNES were included. After receiving written consent, a self-administered questionnaire was given to patients after the attending physician had communicated the diagnosis of PNES. A total of 75 patients were recruited. All patients had their typical seizures recorded on video-EEG (range 1–12, mean 2.18). Seventy patients were satisfied with the diagnosis of PNES. Nine patients did not agree that PNES has a psychological cause. Nineteen patients thought that the EMU doctors had no clue as to the cause of their seizures and 20 thought that there was no hope for a cure of their seizures. A significant number of patients with PNES feel that there is no hope for cure of their seizures. Thorough education about PNES, properly preparing patients before discussing the diagnosis of PNES, and preferably earlier diagnosis may prevent this miscommunication and result in better outcomes. A comprehensive approach including psychological counseling and psychiatric input, evaluation and treatment, in order to bring the illness from the subconscious to the conscious level, and effective follow-up may be helpful. Ó 2016 Published by Elsevier Ltd.
1. Introduction Psychogenic nonepileptic seizures (PNES) are a common cause of refractory seizures. PNES are seen in 10–58% of adult patients with intractable seizures. PNES account for 10–40% of all patients referred to epilepsy centers [1,2]. These seizures are often misdiagnosed and patients are at risk of receiving inappropriate medications for prolonged treatment periods or invasive interventions such as intubation and vagus nerve stimulator implants [3]. Simultaneous video-electroencephalographic (EEG) monitoring has allowed PNES to be effectively diagnosed. Once the diagnosis is established, epileptologists explain to the patients that they do not have epilepsy, but that their seizures are a manifestation of psychological distress. This can be difficult, as patients may be
⇑ Corresponding author. Tel.: +1 615 936 2591. E-mail address: [email protected] (A. Arain).
reluctant to accept the diagnosis. Communicating the diagnosis to the patient is very important as it may have therapeutic implications. A substantial number of patients with PNES become seizure-free shortly after being informed of the diagnosis [4–6]. However if this diagnosis is not accurately perceived by the patients, long term outcome and follow-up in these patients may be compromised. We aimed to evaluate how well we are communicating the diagnosis in our comprehensive epilepsy center.
2. Methods This prospective study was conducted in an eight-bed epilepsy monitoring unit (EMU) where patients were referred for a second opinion for their intractable seizures. We collected basic demographics of the patients in this study (Table 1). Satisfaction with the EMU experience and understanding of the PNES diagnosis were assessed with a self-administered 41 item questionnaire. The first
http://dx.doi.org/10.1016/j.jocn.2015.10.030 0967-5868/Ó 2016 Published by Elsevier Ltd.
Please cite this article in press as: Arain A et al. Communicating the diagnosis of psychogenic nonepileptic seizures: The patient perspective. J Clin Neurosci (2016), http://dx.doi.org/10.1016/j.jocn.2015.10.030
2
A. Arain et al. / Journal of Clinical Neuroscience xxx (2016) xxx–xxx
Table 1 Demographics of patients diagnosed with psychogenic nonepileptic seizures Age, mean (standard deviation) Female/Male
40.5 (12.6) 52/23
Ethnicity Caucasian African American American Indian Marital status Single Married Divorced
9 49 17
Educational status Some schooling High school graduate College Unknown
3 39 31 2
Occupational status Unemployed Employed Home maker Retired Disabled
27 26 3 3 16
72 2 1
27 questions developed by the EMU staff used a Likert scale to analyze the patient’s experience in the EMU. The remaining 14 items evaluated the patient’s understanding of the PNES diagnosis (Table 2) in a yes/no format. Patients were included in the study if their typical seizures were recorded during the video-EEG monitoring, and their seizures were determined to be PNES. The diagnosis of PNES was established when a typical seizure was recorded on video-EEG, found not to be associated with any EEG changes, and was not clinically consistent with epileptic seizures that may occur without scalp EEG changes (such as simple partial seizures, frontal lobe complex partial seizures, or other seizures from a deep source). We excluded patients with coexisting epilepsy, patients with interictal epileptiform discharges and patients in whom the PNES diagnosis was uncertain. All patients who were on antiepileptic drugs had their medications withdrawn when they were admitted to the EMU. Since PNES may coexist with epilepsy in some patients, we performed 3–5 days of video-EEG monitoring after withdrawal of antiepileptic drugs before confirming the absence of coexistent epilepsy.
Table 2 The 14 questionnaire items regarding perception of psychogenic nonepileptic seizures and patient answers Questionnaire item
I am satisfied with the diagnosis of nonepileptic spells. (Q28) Nonepileptic seizures may cause brain damage. (Q29) PNES means psychological cause of seizures. (Q30) Diagnosis will change the outcome of my condition. (Q31) I will follow up with my neurologist. (Q32) I will follow up with neuropsychological testing. (Q33) I will follow up with psychiatrist if referred. (Q34) Doctors have no clue what causes my seizures. (Q35) There is a physical cause for my seizures. (Q36) There is help available to treat my seizures. (Q37) My seizures may improve if underlying stressors are addressed. (Q38) People think that I fake my seizures. (Q39) PNES means being crazy. (Q40) There is no hope for me to get rid of seizures. (Q41)
Patient responses, n Yes
No
Abstained
70
4
1
9
51
15
61 58
8 12
6 5
70 71
4 3
1 1
71 19 32 68 63
3 51 36 4 5
1 5 7 3 7
36 25 20
38 49 52
1 1 3
During the EMU admission, patients were asked whether they would be interested in evaluating the communication of their PNES diagnosis. All patients included in the study signed a written consent form. The study questionnaires were given to the patient by an epilepsy fellow shortly after the attending epileptologist had communicated the final diagnosis of PNES. In our institution, eight attending epileptologists rotated on the EMU. Even though there was no scripted discussion with the patients or their family members, these attendings presented the diagnosis of PNES in a similar way as ‘‘good news” and explained the nature of the condition, its possible causes and the treatment plan. The attending epileptologists also discussed with the patient that discontinuation of antiepileptic drugs avoids unnecessary medication adverse effects and is a key positive aspect of the diagnosis. The treatment plan typically includes neuropsychological testing and counseling in addition to psychiatric evaluation. All patients with the diagnosis of PNES were given a 3 month follow-up appointment in the epilepsy clinic. All patients were also scheduled for neuropsychological testing and referred to a psychiatrist to follow-up as outpatient. All patients with PNES captured during the EMU evaluation were approached for this study. Four patients refused to be enrolled in the study; they did not differ in demographics from other patients. We analyzed 75 consecutive consenting patients diagnosed with PNES on video-EEG monitoring. We also reviewed if patients followed-up in the neurology clinic and if they had neuropsychological testing after discharge from the EMU. In patients who returned for follow-up (33 patients, 44%) we reviewed seizure control at follow-up. The study was approved by the Vanderbilt University Institutional Review Board.
3. Results There were 52 females and 23 males. Their mean age was 40.6 years (range 16–80). The mean age of onset of these seizures was 34.1 years (range 4–76). Patients were monitored for a mean of 3.2 days (range 1–8) in the EMU to capture their typical seizures. The mean duration of PNES before the final diagnosis was 71.5 months (range 1–564 months, median 24 months, standard deviation 123.8 months). The mean duration of PNES in women was 76.1 months (range 1–564 months, median of 24 months and standard deviation 127.2 months) while mean duration of PNES in men was 61 months (range 1–540 months, median 13 months and standard deviation 118.1 months). Males were significantly older than females when they started to have PNES (p = 0.01, Wilcoxon rank sum test) and at the time of video-EEG monitoring (p = 0.02, Wilcoxon rank sum test) (Table 3). Fortytwo patients (56%) had a high school degree and 31 patients (41.3%) had college education while other patients did not finish Table 3 Demographic and clinical variables stratified by sex in patients with psychogenic nonepileptic seizures Variables
Age Age at seizure onset Motionless seizures Motor seizures Duration of seizures (months)
Sex
Patients
Mean
SD
Median
p value (Wilcoxon rank sum)
F M F M F M F M F M
52 23 52 23 52 23 52 23 52 23
38.69 44.74 31.69 39.83 1.35 1.48 2.17 2.13 76.1 61.2
12.69 11.72 14.22 13.68 2.21 1.88 2.56 2.67 127.2 118.1
35.5 49 31 41 1 1 0 1 24 13
0.03 0.01 0.48 0.97 0.06
F = female, M = male, SD = standard deviation.
Please cite this article in press as: Arain A et al. Communicating the diagnosis of psychogenic nonepileptic seizures: The patient perspective. J Clin Neurosci (2016), http://dx.doi.org/10.1016/j.jocn.2015.10.030
A. Arain et al. / Journal of Clinical Neuroscience xxx (2016) xxx–xxx
high school. Psychiatric comorbidities were seen in 42 patients (56%); 19 patients had anxiety and depression, 15 patients reported depression only, three patients reported bipolar disorder, two patients reported anxiety/panic disorder and three patients reported post-traumatic stress disorder. Thirty patients had motor jerking seizures; 25 patients had motionless seizures with unresponsiveness, and 12 patients had jerking in some seizures and remained motionless in other seizures. The mean number of recorded PNES was 1.21. There was a trend for more motor seizures (mean 2.6) than motionless seizures (mean 1.9). Seventy-one patients (95%) answered that they were satisfied with the diagnosis. Educational status was not statistically significant in relation to satisfaction levels (p = 0.88, Fischer’s exact test). Only four patients (5.3%) answered that they were not satisfied with the diagnosis of PNES (one of these patients had a negative attitude towards all questions). Even though 95% of patients answered that they were satisfied with the diagnosis, responses to other items suggested that a substantial number did not accept the diagnosis. Nineteen patients (25.3%) answered that the EMU doctors had no clue as to the cause of their seizures, while six patients (8%) abstained from answering this question. Eight patients (10.6%) did not agree that PNES has a psychological cause. Thirty-two (42.7%) patients believed there was a physical cause for their seizures, but 61 (81.3%) patients also agreed PNES had a psychological cause. Only nine patients (12%) felt that PNES resulted in brain damage; females were significantly more likely to think that PNES resulted in brain damage (p = 0.016, Fischer’s exact). There were 20 patients who were hopeless in terms of ever becoming seizure-free (26%). Sixteen patients (21%) answered that the diagnosis will not change the outcome. Fifty-eight patients (77.3%) felt the diagnosis will change the outcome of their condition. Thirty-six patients (48%) answered that people perceive their seizures as fake. Twenty patients (26.7%) thought there was no hope for cure of their seizures. Twenty-five patients (33.3%) thought a diagnosis of nonepileptic seizures means being ‘‘crazy”. Seventy patients (93%) were willing to follow-up with their neurologist; 33 of these patients (33/70, 47%) actually did so. Among the patients who followed-up in the neurology clinic, only nine (9/33, 27%) were seizure free at the last follow-up. Seventyone patients (94%) reported that they will follow-up for formal neuropsychological testing, but only 11 (11/71, 15.4%) did. Seventy-one patients (94%) were willing to see a psychiatrist if recommended. 4. Discussion PNES are a major reason for refractory seizures. Communicating the diagnosis of PNES to patients can have a significant impact on the outcome and resolution of these seizures [7,8]. Studies have shown that communicating the diagnosis helps in the outcome of the PNES even shortly after the presentation of the diagnosis [6,9–11]. In one study, 27 of 54 PNES patients (50%) were seizure free at 3 months after EMU diagnosis. Among these 27 patients, 24 had already become seizure free upon receiving the diagnosis in the EMU [9]. However, communication of the diagnosis is often difficult and complex, as most patients tend to resist the notion that physical symptoms of PNES are linked to emotional causes [12]. This discussion of the diagnosis between patient and attending epileptologists marks a critical point in the treatment process, heralding a new course of therapy that involves psychiatric evaluation and follow-up [13]. Our study shows that patients with PNES often do not perceive the diagnosis of PNES correctly as was seen in the Carton et al. study [14]. That study was based on telephone interviews after patients were discharged from the EMU [14],
3
while our study used a survey immediately after the final diagnosis was conveyed by the attending physician, so the element of memory loss was eliminated. Despite that, our study demonstrates that there were misunderstandings about the diagnosis in the short term. These misunderstandings about the diagnosis are likely to become greater as time passes when memory loss is likely to impede recollection. Patients’ selective or poor perception of the diagnosis of PNES may reflect underlying denial of the diagnosis or uneasiness in bringing painful emotional trauma to the surface [8,15]. Alternatively, difficulty in understanding the diagnosis may be related to educational background. However, 56% of our patients were college graduates who should have had no difficulty understanding the diagnosis. Denial and regression is a common mechanism of patients to cope with their illness [8,12,15,16]. There was some discordance in our patients’ opinions about certain questions. For example, there were 10 patients who answered that their seizures had a psychological cause but still felt that the doctors had no clue as to the cause of their seizures. Additionally, while 70 of 75 patients initially reported being satisfied with the diagnosis, a quarter of patients also answered that the treating physicians had no clue as to the cause of their illness. This may be consistent with denial and regression. Twenty-six percent of our patients continued to think that the PNES diagnosis would not change their outcome and there was no hope of a cure for their seizures. Studies have shown that diagnosis helps in the outcome of PNES even in as short a time as 24 hours [6]. This difficulty in understanding the diagnosis is likely the reason for poor clinical follow-up for neuropsychological testing and psychiatry appointments. We previously noted that less than one-third (27%) of PNES patients followed-up in the neurology clinic [1]. Denial and regression may explain why some patients continue to have seizures and continue ‘‘doctor shopping” [14]. Moreover patients with PNES often equate a diagnosis of PNES to being mentally ill like patients with psychosis. These patients tend to become defensive (perhaps they are not ready to bear the stress of their psychological conflicts), when the diagnosis of PNES is conveyed [12]. PNES have been seen more frequently in women than men and women tend to have a different perspective than men [17,18]. There were more women than men in our cohort. We found that women are significantly more likely to think that PNES results in brain damage. Men in our cohort were significantly older than women when they started to have PNES and when they presented for video-EEG monitoring (Table 3). This could possibly reflect that men try to suppress their symptoms or work to discover the diagnosis and manage the symptoms on their own and have a stronger defense mechanism of denial [17]. However in our cohort, like other studies, the duration of PNES was not significantly different between the two sexes (p = 0.06, Wilcoxon rank sum test) [17,18]. This study highlights the apparent miscommunication between the patient’s understanding of PNES and the neurologists taking care of these patients. Proper and thorough education along with emotional preparation for an acceptance of a PNES diagnosis may prevent this miscommunication and result in a better outcome. In this regard we can use published strategies to discuss the diagnosis of PNES [4,5,19]. In order to improve the communication of a PNES diagnosis, patients can be given written material about PNES in simplified language [20]. This written material can reinforce the diagnosis when patients read the material at a convenient time. This is a weakness of our study as we did not provide written material. Patients with PNES need further discussion with their referring provider/psychiatrist/psychologist working in conjunction to provide thorough education regarding their PNES diagnosis. In addition, it may not be enough to seek psychiatric follow-up, but
Please cite this article in press as: Arain A et al. Communicating the diagnosis of psychogenic nonepileptic seizures: The patient perspective. J Clin Neurosci (2016), http://dx.doi.org/10.1016/j.jocn.2015.10.030
4
A. Arain et al. / Journal of Clinical Neuroscience xxx (2016) xxx–xxx
rather obtain a psychiatric consultation while the patient is in the EMU. Conflicts of interest/disclosures The authors declare that they have no financial or other conflicts of interest in relation to this research and its publication. References [1] Arain AM, Hamadani AM, Islam S, et al. Predictors of early seizure remission after diagnosis of psychogenic nonepileptic seizures. Epilepsy Behav 2007;11:409–12. [2] Benbadis SR, Allen Hauser W. An estimate of the prevalence of psychogenic non-epileptic seizures. Seizure 2000;9:280–1. [3] Arain AM, Song Y, Bangalore-Vittal N, et al. Long term video/EEG prevents unnecessary vagus nerve stimulator implantation in patients with psychogenic nonepileptic seizures. Epilepsy Behav 2011;21:364–6. [4] Hall-Patch L, Brown R, House A, et al. Acceptability and effectiveness of a strategy for the communication of the diagnosis of psychogenic nonepileptic seizures. Epilepsia 2010;51:70–8. [5] Shen W, Bowman ES, Markand ON. Presenting the diagnosis of pseudoseizure. Neurology 1990;40:756–9. [6] Farias ST, Thieman C, Alsaadi TM. Psychogenic nonepileptic seizures: acute change in event frequency after presentation of the diagnosis. Epilepsy Behav 2003;4:424–9. [7] Karterud HN, Knizek BL, Nakken KO. Changing the diagnosis from epilepsy to PNES: patients’ experiences and understanding of their new diagnosis. Seizure 2010;19:40–6.
[8] Frances PL, Baker GA, Appleton PL. Stress and avoidance in Pseudoseizures: testing the assumptions. Epilepsy Res 1999;34:241–9. [9] Duncan R, Razvi S, Mulhern S. Newly presenting psychogenic nonepileptic seizures: incidence, population characteristics, and early outcome from a prospective audit of a first seizure clinic. Epilepsy Behav 2011;20:308–11. [10] Buchanan N, Snars J. Pseudoseizures (non epileptic attack disorder)–clinical management and outcome in 50 patients. Seizure 1993;2:141–6. [11] Walczak TS, Papacostas S, Williams DT, et al. Outcome after diagnosis of psychogenic nonepileptic seizures. Epilepsia 1995;36:1131–7. [12] Monzoni CM, Duncan R, Grunewald R, et al. How do neurologists discuss functional symptoms with their patients: a conversation analytic study. J Psychosom Res 2011;71:377–83. [13] Harden CL, Ferrando SJ. Delivering the diagnosis of psychogenic pseudoseizures: should the neurologist or the psychiatrist be responsible? Epilepsy Behav 2001;2:519–23. [14] Carton S, Thompson PJ, Duncan JS. Non-epileptic seizures: patients’ understanding and reaction to the diagnosis and impact on outcome. Seizure 2003;12:287–94. [15] Testa SM, Krauss GL, Lesser RP, et al. Stressful life event appraisal and coping in patients with psychogenic seizures and those with epilepsy. Seizure 2012;21:282–7. [16] Thompson R, Isaac CL, Rowse G, et al. What is it like to receive a diagnosis of nonepileptic seizures? Epilepsy Behav 2009;14:508–15. [17] Holmes MD, Dodrill CB, Bachtler S, et al. Evidence that emotional maladjustment is worse in men than in women with psychogenic nonepileptic seizures. Epilepsy Behav 2001;2:568–73. [18] Noe KH, Grade M, Stonnington CM, et al. Confirming psychogenic nonepileptic seizures with video-EEG: sex matters. Epilepsy Behav 2012;23:220–3. [19] Duncan R. Psychogenic nonepileptic seizures: diagnosis and initial management. Expert Rev Neurother 2010;10:1803–9. [20] Thompson NC, Osorio I, Hunter EE. Nonepileptic seizures: reframing the diagnosis. Perspect Psychiatr Care 2005;41:71–8.
Please cite this article in press as: Arain A et al. Communicating the diagnosis of psychogenic nonepileptic seizures: The patient perspective. J Clin Neurosci (2016), http://dx.doi.org/10.1016/j.jocn.2015.10.030
Contents lists available at ScienceDirect
Journal of Clinical Neuroscience journal homepage: www.elsevier.com/locate/jocn
Clinical Study
Communicating the diagnosis of psychogenic nonepileptic seizures: The patient perspective Amir Arain a,⇑, Maamoon Tammaa a, Faria Chaudhary a, Shazil Gill a, Syed Yousuf a, Nandakumar Bangalore-Vittal a, Pradumna Singh a, Shagufta Jabeen b, Shahid Ali b, Yanna Song a, Nabil J. Azar a a b
Department of Neurology, Vanderbilt University Medical Center, A-0118 Medical Center North, Nashville, TN 37232, USA Department of Psychiatry, Meharry Medical College, Nashville, TN, USA
a r t i c l e
i n f o
Article history: Received 27 February 2015 Accepted 4 October 2015 Available online xxxx Keywords: Epilepsy Nonepileptic seizures Pseudoseizures Psychogenic seizures Spells
a b s t r a c t Psychogenic nonepileptic seizures (PNES) are a common cause of refractory seizures. Videoelectroencephalographic (EEG) monitoring has allowed PNES to be effectively distinguished from epileptic seizures. Once the diagnosis of PNES is established, neurologists face the challenge of explaining it to patients. Patients may not always receive the diagnosis well. The aim of this study is to evaluate how effectively patients receive and perceive the diagnosis of PNES. This prospective study was conducted in an eight-bed epilepsy monitoring unit (EMU). Adult patients with newly confirmed PNES were included. After receiving written consent, a self-administered questionnaire was given to patients after the attending physician had communicated the diagnosis of PNES. A total of 75 patients were recruited. All patients had their typical seizures recorded on video-EEG (range 1–12, mean 2.18). Seventy patients were satisfied with the diagnosis of PNES. Nine patients did not agree that PNES has a psychological cause. Nineteen patients thought that the EMU doctors had no clue as to the cause of their seizures and 20 thought that there was no hope for a cure of their seizures. A significant number of patients with PNES feel that there is no hope for cure of their seizures. Thorough education about PNES, properly preparing patients before discussing the diagnosis of PNES, and preferably earlier diagnosis may prevent this miscommunication and result in better outcomes. A comprehensive approach including psychological counseling and psychiatric input, evaluation and treatment, in order to bring the illness from the subconscious to the conscious level, and effective follow-up may be helpful. Ó 2016 Published by Elsevier Ltd.
1. Introduction Psychogenic nonepileptic seizures (PNES) are a common cause of refractory seizures. PNES are seen in 10–58% of adult patients with intractable seizures. PNES account for 10–40% of all patients referred to epilepsy centers [1,2]. These seizures are often misdiagnosed and patients are at risk of receiving inappropriate medications for prolonged treatment periods or invasive interventions such as intubation and vagus nerve stimulator implants [3]. Simultaneous video-electroencephalographic (EEG) monitoring has allowed PNES to be effectively diagnosed. Once the diagnosis is established, epileptologists explain to the patients that they do not have epilepsy, but that their seizures are a manifestation of psychological distress. This can be difficult, as patients may be
⇑ Corresponding author. Tel.: +1 615 936 2591. E-mail address: [email protected] (A. Arain).
reluctant to accept the diagnosis. Communicating the diagnosis to the patient is very important as it may have therapeutic implications. A substantial number of patients with PNES become seizure-free shortly after being informed of the diagnosis [4–6]. However if this diagnosis is not accurately perceived by the patients, long term outcome and follow-up in these patients may be compromised. We aimed to evaluate how well we are communicating the diagnosis in our comprehensive epilepsy center.
2. Methods This prospective study was conducted in an eight-bed epilepsy monitoring unit (EMU) where patients were referred for a second opinion for their intractable seizures. We collected basic demographics of the patients in this study (Table 1). Satisfaction with the EMU experience and understanding of the PNES diagnosis were assessed with a self-administered 41 item questionnaire. The first
http://dx.doi.org/10.1016/j.jocn.2015.10.030 0967-5868/Ó 2016 Published by Elsevier Ltd.
Please cite this article in press as: Arain A et al. Communicating the diagnosis of psychogenic nonepileptic seizures: The patient perspective. J Clin Neurosci (2016), http://dx.doi.org/10.1016/j.jocn.2015.10.030
2
A. Arain et al. / Journal of Clinical Neuroscience xxx (2016) xxx–xxx
Table 1 Demographics of patients diagnosed with psychogenic nonepileptic seizures Age, mean (standard deviation) Female/Male
40.5 (12.6) 52/23
Ethnicity Caucasian African American American Indian Marital status Single Married Divorced
9 49 17
Educational status Some schooling High school graduate College Unknown
3 39 31 2
Occupational status Unemployed Employed Home maker Retired Disabled
27 26 3 3 16
72 2 1
27 questions developed by the EMU staff used a Likert scale to analyze the patient’s experience in the EMU. The remaining 14 items evaluated the patient’s understanding of the PNES diagnosis (Table 2) in a yes/no format. Patients were included in the study if their typical seizures were recorded during the video-EEG monitoring, and their seizures were determined to be PNES. The diagnosis of PNES was established when a typical seizure was recorded on video-EEG, found not to be associated with any EEG changes, and was not clinically consistent with epileptic seizures that may occur without scalp EEG changes (such as simple partial seizures, frontal lobe complex partial seizures, or other seizures from a deep source). We excluded patients with coexisting epilepsy, patients with interictal epileptiform discharges and patients in whom the PNES diagnosis was uncertain. All patients who were on antiepileptic drugs had their medications withdrawn when they were admitted to the EMU. Since PNES may coexist with epilepsy in some patients, we performed 3–5 days of video-EEG monitoring after withdrawal of antiepileptic drugs before confirming the absence of coexistent epilepsy.
Table 2 The 14 questionnaire items regarding perception of psychogenic nonepileptic seizures and patient answers Questionnaire item
I am satisfied with the diagnosis of nonepileptic spells. (Q28) Nonepileptic seizures may cause brain damage. (Q29) PNES means psychological cause of seizures. (Q30) Diagnosis will change the outcome of my condition. (Q31) I will follow up with my neurologist. (Q32) I will follow up with neuropsychological testing. (Q33) I will follow up with psychiatrist if referred. (Q34) Doctors have no clue what causes my seizures. (Q35) There is a physical cause for my seizures. (Q36) There is help available to treat my seizures. (Q37) My seizures may improve if underlying stressors are addressed. (Q38) People think that I fake my seizures. (Q39) PNES means being crazy. (Q40) There is no hope for me to get rid of seizures. (Q41)
Patient responses, n Yes
No
Abstained
70
4
1
9
51
15
61 58
8 12
6 5
70 71
4 3
1 1
71 19 32 68 63
3 51 36 4 5
1 5 7 3 7
36 25 20
38 49 52
1 1 3
During the EMU admission, patients were asked whether they would be interested in evaluating the communication of their PNES diagnosis. All patients included in the study signed a written consent form. The study questionnaires were given to the patient by an epilepsy fellow shortly after the attending epileptologist had communicated the final diagnosis of PNES. In our institution, eight attending epileptologists rotated on the EMU. Even though there was no scripted discussion with the patients or their family members, these attendings presented the diagnosis of PNES in a similar way as ‘‘good news” and explained the nature of the condition, its possible causes and the treatment plan. The attending epileptologists also discussed with the patient that discontinuation of antiepileptic drugs avoids unnecessary medication adverse effects and is a key positive aspect of the diagnosis. The treatment plan typically includes neuropsychological testing and counseling in addition to psychiatric evaluation. All patients with the diagnosis of PNES were given a 3 month follow-up appointment in the epilepsy clinic. All patients were also scheduled for neuropsychological testing and referred to a psychiatrist to follow-up as outpatient. All patients with PNES captured during the EMU evaluation were approached for this study. Four patients refused to be enrolled in the study; they did not differ in demographics from other patients. We analyzed 75 consecutive consenting patients diagnosed with PNES on video-EEG monitoring. We also reviewed if patients followed-up in the neurology clinic and if they had neuropsychological testing after discharge from the EMU. In patients who returned for follow-up (33 patients, 44%) we reviewed seizure control at follow-up. The study was approved by the Vanderbilt University Institutional Review Board.
3. Results There were 52 females and 23 males. Their mean age was 40.6 years (range 16–80). The mean age of onset of these seizures was 34.1 years (range 4–76). Patients were monitored for a mean of 3.2 days (range 1–8) in the EMU to capture their typical seizures. The mean duration of PNES before the final diagnosis was 71.5 months (range 1–564 months, median 24 months, standard deviation 123.8 months). The mean duration of PNES in women was 76.1 months (range 1–564 months, median of 24 months and standard deviation 127.2 months) while mean duration of PNES in men was 61 months (range 1–540 months, median 13 months and standard deviation 118.1 months). Males were significantly older than females when they started to have PNES (p = 0.01, Wilcoxon rank sum test) and at the time of video-EEG monitoring (p = 0.02, Wilcoxon rank sum test) (Table 3). Fortytwo patients (56%) had a high school degree and 31 patients (41.3%) had college education while other patients did not finish Table 3 Demographic and clinical variables stratified by sex in patients with psychogenic nonepileptic seizures Variables
Age Age at seizure onset Motionless seizures Motor seizures Duration of seizures (months)
Sex
Patients
Mean
SD
Median
p value (Wilcoxon rank sum)
F M F M F M F M F M
52 23 52 23 52 23 52 23 52 23
38.69 44.74 31.69 39.83 1.35 1.48 2.17 2.13 76.1 61.2
12.69 11.72 14.22 13.68 2.21 1.88 2.56 2.67 127.2 118.1
35.5 49 31 41 1 1 0 1 24 13
0.03 0.01 0.48 0.97 0.06
F = female, M = male, SD = standard deviation.
Please cite this article in press as: Arain A et al. Communicating the diagnosis of psychogenic nonepileptic seizures: The patient perspective. J Clin Neurosci (2016), http://dx.doi.org/10.1016/j.jocn.2015.10.030
A. Arain et al. / Journal of Clinical Neuroscience xxx (2016) xxx–xxx
high school. Psychiatric comorbidities were seen in 42 patients (56%); 19 patients had anxiety and depression, 15 patients reported depression only, three patients reported bipolar disorder, two patients reported anxiety/panic disorder and three patients reported post-traumatic stress disorder. Thirty patients had motor jerking seizures; 25 patients had motionless seizures with unresponsiveness, and 12 patients had jerking in some seizures and remained motionless in other seizures. The mean number of recorded PNES was 1.21. There was a trend for more motor seizures (mean 2.6) than motionless seizures (mean 1.9). Seventy-one patients (95%) answered that they were satisfied with the diagnosis. Educational status was not statistically significant in relation to satisfaction levels (p = 0.88, Fischer’s exact test). Only four patients (5.3%) answered that they were not satisfied with the diagnosis of PNES (one of these patients had a negative attitude towards all questions). Even though 95% of patients answered that they were satisfied with the diagnosis, responses to other items suggested that a substantial number did not accept the diagnosis. Nineteen patients (25.3%) answered that the EMU doctors had no clue as to the cause of their seizures, while six patients (8%) abstained from answering this question. Eight patients (10.6%) did not agree that PNES has a psychological cause. Thirty-two (42.7%) patients believed there was a physical cause for their seizures, but 61 (81.3%) patients also agreed PNES had a psychological cause. Only nine patients (12%) felt that PNES resulted in brain damage; females were significantly more likely to think that PNES resulted in brain damage (p = 0.016, Fischer’s exact). There were 20 patients who were hopeless in terms of ever becoming seizure-free (26%). Sixteen patients (21%) answered that the diagnosis will not change the outcome. Fifty-eight patients (77.3%) felt the diagnosis will change the outcome of their condition. Thirty-six patients (48%) answered that people perceive their seizures as fake. Twenty patients (26.7%) thought there was no hope for cure of their seizures. Twenty-five patients (33.3%) thought a diagnosis of nonepileptic seizures means being ‘‘crazy”. Seventy patients (93%) were willing to follow-up with their neurologist; 33 of these patients (33/70, 47%) actually did so. Among the patients who followed-up in the neurology clinic, only nine (9/33, 27%) were seizure free at the last follow-up. Seventyone patients (94%) reported that they will follow-up for formal neuropsychological testing, but only 11 (11/71, 15.4%) did. Seventy-one patients (94%) were willing to see a psychiatrist if recommended. 4. Discussion PNES are a major reason for refractory seizures. Communicating the diagnosis of PNES to patients can have a significant impact on the outcome and resolution of these seizures [7,8]. Studies have shown that communicating the diagnosis helps in the outcome of the PNES even shortly after the presentation of the diagnosis [6,9–11]. In one study, 27 of 54 PNES patients (50%) were seizure free at 3 months after EMU diagnosis. Among these 27 patients, 24 had already become seizure free upon receiving the diagnosis in the EMU [9]. However, communication of the diagnosis is often difficult and complex, as most patients tend to resist the notion that physical symptoms of PNES are linked to emotional causes [12]. This discussion of the diagnosis between patient and attending epileptologists marks a critical point in the treatment process, heralding a new course of therapy that involves psychiatric evaluation and follow-up [13]. Our study shows that patients with PNES often do not perceive the diagnosis of PNES correctly as was seen in the Carton et al. study [14]. That study was based on telephone interviews after patients were discharged from the EMU [14],
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while our study used a survey immediately after the final diagnosis was conveyed by the attending physician, so the element of memory loss was eliminated. Despite that, our study demonstrates that there were misunderstandings about the diagnosis in the short term. These misunderstandings about the diagnosis are likely to become greater as time passes when memory loss is likely to impede recollection. Patients’ selective or poor perception of the diagnosis of PNES may reflect underlying denial of the diagnosis or uneasiness in bringing painful emotional trauma to the surface [8,15]. Alternatively, difficulty in understanding the diagnosis may be related to educational background. However, 56% of our patients were college graduates who should have had no difficulty understanding the diagnosis. Denial and regression is a common mechanism of patients to cope with their illness [8,12,15,16]. There was some discordance in our patients’ opinions about certain questions. For example, there were 10 patients who answered that their seizures had a psychological cause but still felt that the doctors had no clue as to the cause of their seizures. Additionally, while 70 of 75 patients initially reported being satisfied with the diagnosis, a quarter of patients also answered that the treating physicians had no clue as to the cause of their illness. This may be consistent with denial and regression. Twenty-six percent of our patients continued to think that the PNES diagnosis would not change their outcome and there was no hope of a cure for their seizures. Studies have shown that diagnosis helps in the outcome of PNES even in as short a time as 24 hours [6]. This difficulty in understanding the diagnosis is likely the reason for poor clinical follow-up for neuropsychological testing and psychiatry appointments. We previously noted that less than one-third (27%) of PNES patients followed-up in the neurology clinic [1]. Denial and regression may explain why some patients continue to have seizures and continue ‘‘doctor shopping” [14]. Moreover patients with PNES often equate a diagnosis of PNES to being mentally ill like patients with psychosis. These patients tend to become defensive (perhaps they are not ready to bear the stress of their psychological conflicts), when the diagnosis of PNES is conveyed [12]. PNES have been seen more frequently in women than men and women tend to have a different perspective than men [17,18]. There were more women than men in our cohort. We found that women are significantly more likely to think that PNES results in brain damage. Men in our cohort were significantly older than women when they started to have PNES and when they presented for video-EEG monitoring (Table 3). This could possibly reflect that men try to suppress their symptoms or work to discover the diagnosis and manage the symptoms on their own and have a stronger defense mechanism of denial [17]. However in our cohort, like other studies, the duration of PNES was not significantly different between the two sexes (p = 0.06, Wilcoxon rank sum test) [17,18]. This study highlights the apparent miscommunication between the patient’s understanding of PNES and the neurologists taking care of these patients. Proper and thorough education along with emotional preparation for an acceptance of a PNES diagnosis may prevent this miscommunication and result in a better outcome. In this regard we can use published strategies to discuss the diagnosis of PNES [4,5,19]. In order to improve the communication of a PNES diagnosis, patients can be given written material about PNES in simplified language [20]. This written material can reinforce the diagnosis when patients read the material at a convenient time. This is a weakness of our study as we did not provide written material. Patients with PNES need further discussion with their referring provider/psychiatrist/psychologist working in conjunction to provide thorough education regarding their PNES diagnosis. In addition, it may not be enough to seek psychiatric follow-up, but
Please cite this article in press as: Arain A et al. Communicating the diagnosis of psychogenic nonepileptic seizures: The patient perspective. J Clin Neurosci (2016), http://dx.doi.org/10.1016/j.jocn.2015.10.030
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rather obtain a psychiatric consultation while the patient is in the EMU. Conflicts of interest/disclosures The authors declare that they have no financial or other conflicts of interest in relation to this research and its publication. References [1] Arain AM, Hamadani AM, Islam S, et al. Predictors of early seizure remission after diagnosis of psychogenic nonepileptic seizures. Epilepsy Behav 2007;11:409–12. [2] Benbadis SR, Allen Hauser W. An estimate of the prevalence of psychogenic non-epileptic seizures. Seizure 2000;9:280–1. [3] Arain AM, Song Y, Bangalore-Vittal N, et al. Long term video/EEG prevents unnecessary vagus nerve stimulator implantation in patients with psychogenic nonepileptic seizures. Epilepsy Behav 2011;21:364–6. [4] Hall-Patch L, Brown R, House A, et al. Acceptability and effectiveness of a strategy for the communication of the diagnosis of psychogenic nonepileptic seizures. Epilepsia 2010;51:70–8. [5] Shen W, Bowman ES, Markand ON. Presenting the diagnosis of pseudoseizure. Neurology 1990;40:756–9. [6] Farias ST, Thieman C, Alsaadi TM. Psychogenic nonepileptic seizures: acute change in event frequency after presentation of the diagnosis. Epilepsy Behav 2003;4:424–9. [7] Karterud HN, Knizek BL, Nakken KO. Changing the diagnosis from epilepsy to PNES: patients’ experiences and understanding of their new diagnosis. Seizure 2010;19:40–6.
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Please cite this article in press as: Arain A et al. Communicating the diagnosis of psychogenic nonepileptic seizures: The patient perspective. J Clin Neurosci (2016), http://dx.doi.org/10.1016/j.jocn.2015.10.030